A 38-year-old woman with necrotising cervical lymphadenitis due to Histoplasma capsulatum

Vosse, Esther van de; Wengen, Annelies van; Meide, Wendy F. van der; Visser, Leo G.; Dissel, Jaap T. van

doi:10.1007/s15010-017-1060-x

Cited by 16 publications

(14 citation statements)

References 22 publications

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“…Other opportunistic infections developed within 1 year after occurrence of disseminated NTM infection. 4 One patient had pulmonary tuberculosis, the other patient had tuberculosis pleurisy.…”

Section: Discussionmentioning

confidence: 99%

Incorrect diagnoses in patients with neutralizing anti-interferon-gamma-autoantibodies

Wang

Sheng

et al. 2020

Clinical Microbiology and Infection

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Disseminated non-tuberculous mycobacterial infections Incorrect diagnosesNeutralizing anti-interferon-gammaautoantibodies TB metastatic carcinoma a b s t r a c t Objectives: Early diagnosis of adult-onset immunodeficiency associated with neutralizing antiinterferon-gamma autoantibodies (anti-IFNg Abs) remains difficult given the lack of a distinctive phenotype and a routine test. This study aimed to investigate the determinants of incorrect tentative diagnoses and useful clues for early disease recognition. Methods: This study enrolled adult patients who had unexplained opportunistic infections diagnosed at six hospitals and identified those having neutralizing anti-IFNg Abs (cases). Demographics, medical history, initial presentations and laboratory data, causative pathogens, tentative diagnoses, and treatment were analysed and compared among individuals having neutralizing anti-IFNg Abs (cases) and those without (controls). Results: Among the 154 patients enrolled, neutralizing anti-IFN-g Abs were detected in 50 (71%) of 70 patients with disseminated non-tuberculous mycobacterial infection (dNTM) but not in 84 patients without dNTM. The median time from disease onset to the recognition of dNTM associated with neutralizing anti-IFNg Abs was 1.6 years (range, 0.25e19 years). Incorrect tentative diagnoses resulted in the administration of anti-tuberculosis regimens (60%, 30/50), immunosuppressants (48%, 24/50), and systemic chemotherapy (2%, 10/50) to the 50 cases. Multivariate analysis revealed that case patients were more likely than controls to present with multiple bone lesions (adjusted odds ratio (OR), 27.16; 95% confidence interval (CI), 1.21e609.59) and leukocytosis (adjusted OR, 1.48; 95% CI, 1.12e1.95); however, the controls had a higher rate of mycobacterial bloodstream infection (adjusted OR, 0.05; 95% CI 0.00 e0.66). Conclusions: The high rate of incorrect tentative diagnoses led to frequent inappropriate management in patients with neutralizing anti-IFNg Abs, and highlighted the need for increased awareness among clinicians. U.-I.

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Section: Discussionmentioning

confidence: 99%

Incorrect diagnoses in patients with neutralizing anti-interferon-gamma-autoantibodies

Wang

Sheng

et al. 2020

Clinical Microbiology and Infection

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“…The patient was previously treated for an angioimmunoblastic T-cell lymphoma with complete remission [13]. Van de Vosse et al [14] reported nAIFNG in a 38–year-old women of African descent, who presented with necrotizing lymphadenitis due to Histoplasma capsulatum . Her HLA haplotype did not overlap with the one found to be associated with AIGA [10,14].…”

Section: Autoantibodies To Ifn-γ Cause Susceptibility To Opportunistimentioning

confidence: 99%

“…Van de Vosse et al [14] reported nAIFNG in a 38–year-old women of African descent, who presented with necrotizing lymphadenitis due to Histoplasma capsulatum . Her HLA haplotype did not overlap with the one found to be associated with AIGA [10,14]. Miyashita et al [15] describe nAIGA associated disseminated M. avium infection presenting as bladder lesions and sterile pyuria in a 63-year-old Japanese women.…”

Section: Autoantibodies To Ifn-γ Cause Susceptibility To Opportunistimentioning

confidence: 99%

Anticytokine autoantibodies leading to infection: early recognition, diagnosis and treatment options

Barcenas-Morales

Cortes-Acevedo

Döffinger

2019

Current Opinion in Infectious Diseases

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Purpose of review The current review gives a concise and updated overview of the relative new field of anticytokine autoantibodies (ACAA) and associated infections with a focus on recent findings regarding clinical manifestions, diagnostic and treatments. Recent findings Several recent case reports of unusual presentations of patients with neutralizing autoantibodies to IFN-γ and granulocyt macrophage colony-stimulating factor and expand the spectrum of clinical manifestations and suggest that anticytokine-mediated acquired immunodeficiency causing susceptibility to infection may be underdiagnosed. There is an expanding geographical distribution of antigranulocyt macrophage colony-stimulating factor associated Cryptococcus gattii infection. The spectrum of identified infections in patients with neutralizing antibodies to IFN-γ has a strong endemic component. Rituximab or cyclophophamide in addition to antimycobacterials could be a treatment options in refractory cases. NF-κB2 deficiency may be associated with a complex pattern of high titre neutralizing ACAA similar to autoimmune polyglandular syndrome type I and Thymoma. New technique for the detection of anticytokine antibodies are presented. Quantiferon testing, which is widely available for TB-diagnostic, may be repurposed to detect anti-IFN-γ autoantibodies. We propose that this test could be as well used to show if they are neutralizing. Summary ACAA are an emerging cause of acquired immunodeficiency which is likely underdiagnosed. Recent case reports document expanding spectra of clinical manifestations. NF-κB2 deficiency may be associated with a complex anti cytokine autoantibody pattern.

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“…32–34 However, opportunistic pathogenic infections have been reported in Caucasian patients with anti-IFN-γ AAbs living in the UK, USA, Germany as well as in patients of African ancestry. 16,19,20,35 Although correlations between anti-IFN-γ AAbs, types of opportunistic infections, clinical manifestations, HLA alleles, and ethnicity have been demonstrated, other issues, such as environmental factors and genetic diversity must be elucidated for in-depth understanding of this disease. Interestingly, by fitting a linear mixed model adjusted for age, sex, as well as the presence of infection, antibiotic use and cyclophosphamide or rituximab use, anti-IFN-γ AAbs levels decreased overtime regardless of ethnicity, even in the absence of immunomodulatory therapy.…”

Section: Anti-ifn-γ Aabs and Pathologymentioning

confidence: 99%

Minireview: Insights into anti-interferon-γ autoantibodies

Chawansuntati

Rattanathammethee

Wipasa

2021

Exp Biol Med (Maywood)

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The association between the presence of anti-interferon-γ autoantibodies and the onset of immunodeficiency with intracellular infections has been clearly established. No standard regimen to control the production of these pathogenic autoantibodies, apart from antimicrobial therapy to eliminate infections, contributes to the medical burden of this syndrome, which sometimes has a fatal outcome. In this review, we summarize the findings on anti-interferon-γ autoantibodies to facilitate further research and to provide guidance for treatment strategies.

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A 38-year-old woman with necrotising cervical lymphadenitis due to Histoplasma capsulatum

Cited by 16 publications

References 22 publications

Incorrect diagnoses in patients with neutralizing anti-interferon-gamma-autoantibodies

Incorrect diagnoses in patients with neutralizing anti-interferon-gamma-autoantibodies

Anticytokine autoantibodies leading to infection: early recognition, diagnosis and treatment options

Minireview: Insights into anti-interferon-γ autoantibodies

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